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Fourth branchial pouch sinus:
Diagnosis and treatment
RICHARD M. ROSENFELD, MD, and HUGH F.BILLER, MD, Pittsburgh. Pennsylvania, and New York, New York
The fourth branchial pouch sinus (FBPS) Is a rare translaryngeal anomaly with diverse
manifestations. Including neonatal stridor and recurrent deep neck Infection. Review
of the world literature reveals 23 reports of sinuses consistent with fourth pouch origin.
We present two additional cases, Including the only example of a right-sided FBPS.
Retrograde excision, beginning at the piriform apex, ensurescomplete removal of the
tract and protection of the recurrent nerve. Theposterior border of the thyroid ala must
be resected or retracted for adequate exposure. Failure to remove the translaryngeal
portion of the tract almost guarantees recurrence. (OTOLARYNGOL HEAD NECK SURG 1991;
105:44,)
METHODS
The branchial pouches of the primitive pharynx are the
endodermal companions to the four ectodermal clefts
first noted in the human embryo by Von Baer in 1827. I
After penetrating the surrounding mesoderm, the elongated pharyngobranchial ducts of the third and fourth
pouches quickly lose their pharyngeal connections." Incomplete obliteration results in a vestigial sinus of the
piriform fossa. with a predictable origin and course
determined by the fixed mesodermal derivatives of adjacent arches.' Clinical experience suggests that persistence of the fourth pouch is a rare, but well-defined,
clinical entity that offers conceptual, diagnostic, and
therapeutic challenges not encountered with anomalies
of more rostral arches."!'
Review of the "branchial" literature, however, reveals
that a great deal of confusion sHU exists regarding sinuses of the third, and especially the fourth branchial
pouches. The purpose of this article is to present a lucid
description of the fourth branchial pouch sinus (FBPS)
on the basis of surgical experience with two patients,
and a review of the world literature and pertinent embryology. Distinguishing features from the more common second and third arch anomalies are discussed.
The value of developmental anatomy as a surgical
"road-map" in a potentially amorphous mass of postinflammatory fibrosis is emphasized.
From the Department of Pediatric Otolaryngology, Children's Hospital of Pittsburgh (Or. Rosenfeld), and the Department of 010laryngology, Mount Sinai Medical Center (Or. Biller).
Presented at the Annual Meeting of the Triological Society.
New Haven, Conn., Jan. 26-27, 1990.
Received for publication May 14, 1990; accepted Feb. II. 1991.
Reprint requests: Richard M. Rosenfeld. MD, Department of Pediatric Otolaryngology, Children's Hospital of Pittsburgh. 3705 Fifth
Avenue. Pittsburgh. PA 15213-2583.
•
2311128883
The distinction between sinuses of the third and
fourth branchial pouches is not possible using clinical
and/or radiographic criteria because both sinuses originate from the piriform fossa and have similar clinical
presentations, Consequently, only surgically explored
sinuses reported in sufficient detail to be suggestive of
fourth pouch origin have been included in this review
(Tables I and 2). Possible remnants of the fourth branchial cleft, lacking a piriform connection, have been
reported by Downey and Ward l 4 and Shugar and Healy"
and will not be considered in this review.
Among these 23 patients are eight classified by Miyauchi et al." as third pouch sinuses, despite surgical
findings consistent with a diagnosis of FBPS. The authors base their diagnosis on the observations that the
superior parathyroid gland (pouch 4) was consistently
unrelated to the observed sinus, and some patients had
ectopic thymus (pouch 3) adjacent to the tract; however,
pharyngobranchial duct remnants do not necessarily involve their respective parathyroids, and thymic tissue
may originate from the fourth pouch. 16 Because remnants of third and fourth pouch glandular structures, as
well as thyroid tissue, have been reported in relation
to the FBPS tract, 12 histologic findings should be viewed
with caution as indicators of arch derivation.
CASE REPORTS
Case 1, A 31/2-year-old boy had a I-year history of recurrent acute suppurative thyroiditis. Incision and drainage
suggested a pharyngeal connection, because liquids subsequently dribbled from the site during meals. Barium swallow
revealed a slender sinus originating from the right piriform
apex (Fig. I), and a diagnosis of third or fourth pouch sinus
was entertained.
Surgical explorationdemonstrateda fistulacommencingat
the anterior border of the midportion of the sternomastoid
Volume 105 Number 1
July
Fourth branchial pouch sinus: Diagnosis and treatment
1991
lablei.
a
Criteria for diagnosis of fourth branchial pouch sinus
Orlgln
Course
Termination
Connection with piriform fossa
must be demonstrated by
radiography. endoscopy. or
surgical exploration
Surgery must reveal tract which
descends from piriform passIng caudal to SLN and rostal
to RLN
Physical examination, radiography
or surgery must show tract
ending as a blind sinus. cyst.
abscess. or fistula within or adjacent to superior pole of thyroid gland
SLN. Superior laryngeal nerve; RLN. recurrent laryngeal nerve.
lable 2. Surgically explored
piriform apex sinuses consistent with a diagnosis of fourth branchial
pouch sinus
Patient
no.
Sex
1
2
M
3
?
4
5
M
6
7
8
9
10
11
12
13
14
15
16
17
18
19
20
21
22
23
24
25
F
F
SouIce lyear)
Sandborn' (1972)
Tucker and Skolnick"
(1973)
Roediger et al.? (1977)
9 mo/2 mo
5 yr/6 mo
Andrieu-Guitrancourt
et al.'o (1979)
30 d/30 d
M
F
F
Mlyauchi et al.9 (1981)
F
F
M
F
M
F
F
M
F
M
M
M
F
F
F
M
M
Age at
dlagnosls/on..t
IYI/mo/d)
Burge 3 (1983)
Ostfeld et al.' (1985)
Tovi et al.8 (1985)
Kartheuser et al.6 (1986)
Tanyel et al." (1986)
Andrieu-Guitrancourt
et al.'2 (1988)
Rosenfeld and Biller (1989)
3 d/3 d
Birth/Birth
Birth/Birth
6 yr/6 yr
18yr/6yr
17 yr/10 yr
7 yr/7 yr
12 yr/4 yr
B yr/B yr
13 yr/8 yr
50 yr/l0 yr
55 yr/7 yr
6mo/Birth
15 yr/13 yr
1 yr/?
9 yr/1 yr
7 mol?
17 yr/17 yr
14 yr/?
21 yrl 18 yr
3 yr/2 yr
17 yr/14 yr
Side; presentation
No. ot I.D/exclalon
L-LNC; hoarseness
L-LNA, recurrent
0/1
1/1
L-LNC; impending respiratory
distress
L-LNA; cyanosis
L-LNC; impending respiratory
distress
L-LNC; stridor
L-AST, recurrent
L-AST, recurrent
L-AST. recurrent
L-AST, recurrent
L-AST, recurrent
L-AST, recurrent
L-AST. recurrent
L-AST, recurrent
L-AST. recurrent
L-LNC; stridor
L-LNA, recurrent
L-AST, recurrent; PCF
L-LNA, recurrent
L-LNA; PCF
L-AST
L-AST, recurrent
L-AST, recurrent
R-AST, recurrent; PCF
L-LNC, recurrent; PCF
0/2
3/3
1/2
0/1
1/3
All patients
had between
one and ten
episdoes
before
referral for
treatment
1/1
1 11
2/1
2/2
1/1
Oil
3/2
0/2
2/2
L, Left; R, right; LNC, lateral neckcyst; LNA, lateral neckabscess; AST, acute suppurative thyroiditis; PCF, pharyngocutaneous fistula; 1&0. incision
and drainage.
Table 3. Mesodermal
-
derivatives of the branchial arches
Thirdarch
Fourth arch
Sixth arch
Glossopharyngeal nerve
Hyoid body and greater cornu
Common carotid artery
Proximal internal carotid artery
Superior constrictor muscle
Superior laryngeal nerve
Thyroid cartilage
Aortic arch
Right subclavian artery
Inferior constrictor muscle
Recurrent laryngeal nerve
Cricoid and arytenoid cartilages
Ductus arteriosus
Intrinsic laryngeal muscles
46
otolaryngologvHead and Neck Surgery
ROSENFELD and BILLER
Fig. 1. Barium swallow demonstrating slender sinus orig inating from the right p iriform apex (arrow) .
muscle. adherent to the superior pole of the thyroid gland .
The tract entered the larynx after passing under the inferior
constrictor muscle near the cricothyroid joint. Postinflammatory fibrosis prevented ident ification of the recurrent laryngeal nerve (RLN); hence. the tract was ligated cxtralaryngeal and the procedure terminated . Histology showed a
squamous-lined cyst and tract . and a diagnosis of fourth branchial pouch sinus was made .
Recurrent deep neck abscess after 2 month s neces sitated
re-exploration. A 3- x 3-cm abscess cav ity was encountered
adherent to the ala of the thyroid cartilage . Separation of the
cricothyroid joint and excision of a I-em strip of posterior
thyroid ala ( Fig. 2) revealed a tract originating from the pirifonn apex. extending caud ally for 2 em. then turning lateral
to exit the larynx at the cricothyroid joint (Fig . 3). The tract
was ligated at its piriform origin. inferior to the superior
laryngeal nerve (SLN) . and excised. Identification of the RLN
was not possible becau se of extensive scarring . Histolog y
showed a squamous-lined tract with islands o f ectopic thyroid
tissue .
Case 2. A 17-year-old boy had a 3-year history of recurrent
anterolateral left neck cyst and fistula. of presumed thyroglossal origin. Three earlier attempts at exc ision were unsuccessful. A pharyngeal connection was evident from the
appearance of liquids in the neck immediately after drinking .
Barium swallow revealed a left piriform apex sinus tract.
Exploration demonstrated a fistula commencing anterior to
the lower third of the sternomastoid muscle . passing medial
to the carot id sheath toward the cricothyroid joint. superfi cial
to the RLN . A Woodman approach with disarticulation of the
cricothyroid joint and anterior retraction of the thyroid ala
exposed the origin from the piriform apex. inferior to the
SLN . The 6-cm tract was completely exci sed . with pursestring clo sure of the pharyngeal defect. Histology revealed a
squamous lining .
DISCUSSION
Fundamental to an under standing of the fourth bran chial pouch anomaly is a clear distinction between the
terms cyst. sinus. and fi stula. Cysts may occur independently. or in association with a sinus or fistula. A
sinus is a tract that maintains a connection with either
the skin (branchial cleft sinus) or the pharynx (branchial
pouch sinus). " Generally. the branchial cleft sinus is
associated with rostral arches (first and second). and
the branchial pouch sinus with caudal arches (third and
fourth). Intermediate arche s (second and third) can also
form fistulae. or tracts joining the pharynx and skin. A
true fistula of the fourth arch. although theoretically
possible. would follow an extremely tortuous course.
and has never been reported. A pseudo-fistula. however.
may develop after iatrogenic or spontaneous rupture
Volume 105 Number 1
July 1991
Fourth branchial pouch sinus: Diagnosis and treatment 47
Fig. 2. Cyst cavi1y and the extralaryngeal portion of the tract have been removed to expose the
thyroid ala and cricothyroid joint. Arrow indicates position at which tract entered larynx; arrowheads
point to proposed thyroid cartilage cut.
of a cervical abscess caused by a branchial pouch
sinus.
Branchial pouch sinuses may arise from the second
through fourth arches (Fig. 4); the first pouch persists
as the tubotympanic recess, and the rudimentary sixth
POuch-actually an appendage of the fourth-lacks an
independent pharyngeal connection.' The origin of the
seCond pouch sinus from the tonsillar fossa, and its
course between the internal and external carotid arteries, is well-known to clinicians from its frequent occurrence as the internal component of the second arch
fistula. Less commonly appreciated is the ability of an
isolated second pouch sinus to cause recurrent unilateral
tonsillitis and parapharyngeal abscesses." Confusion
regarding the third and fourth pouch sinuses stems from
their common piriform fossa origin and similar clinical
presentations as neck or thyroid abscesses.':" Distinction, however, is of more than academic interest because of the unique surgical approach required for the
fourth pouch sinus.
A third branchial pouch sinus must pass between
mesodermal derivatives of the third and fourth arches
(Table 3). Thus the tract would exit from the rostral
aspect of the piriform sinus, pierce the thyrohyoid membrane cranial to the SLN and inferior constrictor, then
descend between the common carotid artery and vagus
nerve, ending lateral to the thyroid gland." Likewise,
a sinus from the fourth pouch must pass between fourth
and sixth arch structures (there is no fifth arch in human
beings). After originating near the piriform apex, cau-
41
ROSENFELD and BillER
OtolaryngologyHead and Neck Surgery
FIg. 3. Removal of posterior aspect of thyroid cartilage clearly reveals tract origin from piriform apex.
below superior laryngeal nerve. Arrows indicates facet of cricothyroid joint; arrowhead shows cut
edge of thyroid 010.
dal to the SLN, the tract would descend translaryngeal
under the thyroid ala to emerge beneath the inferior
constrictor muscle, and exit the larynx near the cricothyroid joint. Descending superficial to the RLN. the
tract would terminate paratracheal, or within the thyroid
gland (Fig . 5).12.21
Table 2 shows that the FBPS is a unilateral disorder
with a slight female predominance, reported in patient s
from birth to 55 years, with onset of symptoms occurring before age 20 years (mean age , 6 years). This
contrasts with the traditional description of branchial
sinuses, as almost always seen at or shortly after birth
and occurring bilaterally in up to one third of those
affected. 14 In some patients, the FBPS appears capable
of remaining quiescent for years, or even decades , despite initial episodes in childhood or adolescence (patients 14 and 15).
All patients , except one (patient 24) , had a left-sided
sinus. Possible explanations include diminished or absent growth of the right ultimobranchial body/ the ob-
servation of a similar left-sided predominance of congenital thymic anomalies;" and the asymmetric development offourth arch vascular structures . 12 Conversely,
right-sided sinuses may simply demonstrate a greater
propen sity to remain asymptomatic .
Two clinical presentations are apparent from an examination of Table I. The first consists of neonates
with a lateral neck cyst or abscess associated with actual, or impending, airway compromise (patients 3
through 6 and 16). The mass mimics a cystic hygroma.
and may contain air or increase in size during crying
or Valsalva . A second presentation is that of a child.
adolescent. or occasionally an adult, with recurrent infections of the neck or thyroid gland. All patients have
a demonstrable internal sinus tract originating at the
piriform apex; a pharyngocutaneous fistula is not uncommonly seen after external drainage, either spontaneous or surgical, of an acutely infected sinus (patients
18,20,24, and 25).
Complete surgical excision of the tract, including its
Volume 105 Number 1
July 1991
Fourth branchial pouch sinus: Diagnos is and treatment
49
4 th POU CH
UN --4r.~
Fig. 5. Posterior-oblique view demonstrates translaryngeal
c ourse of fourth bran c hial pouch sinus. Forclar i1y. piriform fossa
ha s been rem oved . except for a po rtion of the apex . Position
of tract corresponds to embryologic descent of parathyroid
N via phoryngobronchiol duct.
Fig . 4 . Comparison of the origin and course of vestigial sinuses
of the second. third. and fourth b ranch ial pouches.
piriform attachment. is the most effective treatment of
the FBPS. This is possible only if the disorder is suspected preoperatively and appropriate exposure of the
piriform fossa is planned: partial excision carries a high
risk of recurrence (see case I). Every patient matching
one of the two clinical presentations described earlier
should have an active search for a piriform apex sinus
tract before surgery. If a barium swallow or sinogram
is nondiagnostic. direct hypopharyngoscopy should be
performed at the time of surgical excision. When direct
examination fails to reveal an obvious sinus. gentle
external pressure on the neck may produce a reflu x of
pus or fluid into the piriform. Because all patients reviewed had a sinus demonstrated by one of these methods. the failure to demonstrate a sinus argues strongly
against the diagnosis of FBPS.
Acutely infected sinuses are best treated with antibiotics. and incision and drainage if necessary. Definitive excision should be delayed for several weeks. until
inflammation has completely resolved. A surgical approach beginning with exposure of the thyroid ala and
carotid sheath allows the operation to commence in a
region relatively free of postinflammatory fibrosis. and
permits immediate distinction between branchial pouch
sinuses of the third and fourth arches. If a tract is dis-
10 ROS£NFEl.D and BUER
covered exiting from the thyrohyoid membrane, superior to the SLN, passing between the vagus and common carotid artery. the diagnosis of third pouch sinus
is confirmed, and exposure of the piriform fossa is not
required. Absence of a tract or fibrosis in this region
suggests a FBPS, and the piriform apex must be
exposed by a Woodman approach," or preferably
by excision of a vertical strip of posterior thyroid ala
( Fig. 3) after first disarticulating the cricothyroid joint.
The FBPS tract is ligated from the piriform and any
pharyngeal defect repaired with purse-string closure.
Retrograde excision is then performed, ending with a
surrounding ellipse of skin if a fistula was present. A
portion of the superior pole of the thyroid gland may
be included if necessary; the superior parathyroid
should be preserved. If the tract descends paratracheal,
exposure of the RLN is necessary to prevent injury (a
transient postoperative vocal cord paralysis developed
in patient 3). When scarring or inflammation prevent
adequate identification of the RLN, the excision should
terminate at the cricothyroid region to prevent accidental nerve injury. Once the piriform connection is completely severed, there appears little tendency for recurrence.
CONCWlIONI
1. The FBPS is a well-defined clinical entity that
must be considered in the differential diagnosis
of neonatal respiratory distress, and recurrent
deep neck infection in children and adults.
2. All patients have a demonstrable internal sinus
from the piriform apex by contrast radiography
or endoscopy. Surgical exploration is necessary
to distinguish a FBPS from the more common
sinus of the third pouch.
3. Excision is best performed retrograde, beginning
with complete exposure of the piriform fossa; removal of only the extralaryngeal portion of the
tract almost guarantees recurrence.
4. A high index of suspicion, combined with a thorough understanding of the developmental anatomy of the branchial region, should lead to earlier
detection and more effective treatment.
REFERENCES
I. Meyer HW. Congenital cysts and fistulae of the neck. Ann Surg
1932;95: 1-26. 226-48.
OtolaryngologyHead and Neck SUrgery
2. Davies J. Embryology and anatomy of the head, neck, face,
palate, nose, and paranasal sinuses. In: Paparella MM, Shumrick.
DA, eds. Otolaryngology. Philadelphia: WB Saunders Co., 1980:
(Part I) 63-123.
3. Burge D. Middle ton A. Persistent pharyngeal pouch derivations
in the neonate. J Pediatr Surg 1983;18:230-4.
4. Sandborn WD. A branchial cleft cyst of fourth pouch origin.
J Pediatr Surg 1972;7:82.
5. Tucker HM, Skolnick. ML. Fourth branchial cleft (pharyngeal
pouch) remnant. Trans Am Acad Ophthalmol Otolaryngol
1973;TI:368-71.
6. Kartheuser A, Claus D, Gosseye S, Otte JB. Third and fourth
branchial fistulas. Chir Pediatr (Paris) 1986;27:50-2.
7. Ostfeld E, Segal J. Auslander L. Rabinson S. Fourth pharyngeal
pouch sinus. Laryngoscope 1985;9S:1I14-7.
8. Tovi F, Gatot A, Bar-Ziv J, Yanay I. Recurrent suppurative
thyroiditis due to fourth branchial pouch sinus. Int J Pediatr
Otorhinolaryngol 1985;9:89-96.
9. Miyauchi A. Matsuzuka F, Takai S, Kuma K. Kosaki G. Piriform
sinus fistula. Arch Surg 1981;116:66-9.
10. Andrieu-Guitrancourt J. Narcy P, Borde J, et al. Digestive duplications affecting the pharynx or fistulae of the fourth endobranchial pouch. Four cases. Ann Oto-Laryngol (Paris) 1979;
96:863-79.
11. Tanye\ FC. Cakmak 0, Caglar M, Balkanci F. Fourth pharyngeal
pouch remnant presented as a neck mass. Z Kinderchir
1986;41:360-1.
12. Andrieu-Guitrancourt J, Amstutz I, Buffet X. Bui P, Dehesdin
D. Recurrent lateral cervical suppuration. Role of fistulae and
cysts of fourth branchial pouch. Ann Oto-Laryngol (Paris) 1988;
105:189-92.
13. Roediger WEW, Kalk F. Spitz L. Schmaman A. Congenital
thyroid cyst of ultimobranchial gland origin. J Pediatr Surg
1977;12:57S-6.
14. Downey WL, Ward PH. Branchial cleft cysts in the mediastinum.
Arch Otolaryngol 1969;89:762-5.
IS. Shugar MA. HealyGB. The fourth branchial cleft anomaly. Head
Neck Surg 1980;3:72-5.
16. Zarbo RJ. McClatchey KD, Areen RG, Baker SB. Thymopharyngeal duct cyst. A form of cervical thymus. Ann Otol Rhinol
Laryngol 1983;92:284-9.
17. Chandler JR, Mitchell B. Branchial cleft cysts. sinuses and fistulas. Otolaryngol Clin North Am 1981;14:175-86.
18. Boysen ME. DeBesche A, Djupesland G, Thorud E. Internal
cysts and fistulas of branchial origin. J Laryngol Otol 1979;
93:533-9.
19. Miller 0, Hill JL, Chen-Chih S. O'Brien DS, Haller JA Jr. The
diagnosis and management of pyriform sinus fistulae in infants
and young children. J Pediatr Surg 1983;18:377-81.
20. Reiter D. Third branchial cleft sinus: an unusual cause of neck
abscess. Int J Pediatr Otorhinolaryngol 1982;4:181-6.
21. Liston SL. Fourth branchial fistula. Head Neck Surg 1981;
89:520-2.
22. Woodman G. A modification of the extralaryngeal approach to
arytenoidectomy for bilateral abductor paralysis. Arch Otolaryngol 1946;43:63-S.
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